Non-alcoholic steatohepatitis (NASH) and oral lichen planus: a rare occurrence

Abstract

Oral lichen planus (OLP) is frequently associated with hepatitis C virus infection but uncommonly with other causes of liver disorder. The authors report the case of a 41-year-old male patient with a clinical and histological diagnosis of OLP who presented with a marked alteration of the transaminase values, with no signs of past or present HBV, HCV, HGV or TTV infection. The patient did not consume alcohol and no exposure to hepatotoxic substances was reported. All autoantibodies were negative. Hepatic fine needle biopsy showed macrovesicular steatosis with a slight chronic portal inflammatory infiltrate and signs of siderosis. Iron metabolism was slightly altered. Genetic tests showed a heterozygotic mutation for hereditary haemochromatosis gene (HLA-H C282Y) but not for HLA-H63D. The patient presented slight insulin resistance but had normal glycaemic values. The results are consistent with a diagnosis of non-alcoholic steatohepatitis (NASH). This is the first reported case of NASH associated with OLP.

Lichen planus (LP) is a chronic inflammatory disease that affects skin and mucous membranes of squamous cell origin . In most instances, cutaneous lesions of LP are self-limiting and cause itching, however oral lesions in LP (OLP) are chronic, rarely undergo spontaneous remission, are potentially premalignant and often a source of morbidity . Oral lesions, unlike cutaneous lesions, are difficult to palliate .

A large body of evidence supports a role for immune dysregulation in the pathogenesis of LP, specifically involving the cellular arm of the immune system . LP is probably a stereotype cell mediated reaction to a variety of extrinsic antigens, altered self-antigens, or super antigens. Among the extrinsic factors, several infective agents including some viruses and Helicobacter pylori have been linked to LP, but sometimes on the basis of equivocal data .

A possible link between hepatitis viruses and LP has been suggested by the fact that LP has been frequently associated with chronic liver disease (CLD) in Mediterranean but not in Northern European patients . The hepatitis C virus (HCV) is the main cause of liver disease in OLP patients and the association is mainly evident in Japan and Mediterranean countries . There are a few reports of mainly skin lichenoid eruptions following administration of different hepatitis B virus (HBV) vaccines but most patients with LP and CLD are not HBV-infected and the recently discovered viruses, hepatitis G virus (HGV) and transfusion transmitted virus (TTV), are not often associated with LP . Other hepatic conditions such as Wilson’s disease, haemochromatosis, primary sclerosing cholangitis and alpha-1-antitrypsin deficiency have rarely been related to LP and the association of LP with primary biliary cirrhosis is mostly due to the administration of penicillamine treatment .

The authors report the first case of non-alcoholic steatohepatitis (NASH) in an Italian patient with OLP.

Case report

A 41-year-old male patient was referred in 2005 for a suspected OLP. Oral examination revealed bilateral reticular/atrophic lesions of OLP on the buccal mucosae ( Fig. 1 A and B ). The unremarkable medical and drug history and the lack of any amalgam restorations close to the lesions ruled out the possible diagnosis of oral lichenoid eruptions. Oral biopsy confirmed the clinical diagnosis of OLP. The haematoxylin–eosin stained section showed hyperorthokeratosis of the squamous epithelium, evidence of basal cell damage and a dense band-like lympho-histiocytic infiltrate at the epithelio–mesenchymal junction ( Fig. 2 A ). Immunohistochemical analysis of the inflammatory component demonstrates a predominance of T lymphocytes ( Fig. 2 B) with scattered B lymphocytes ( Fig. 2 C) and very few plasma cells ( Fig. 2 D).

Fig. 1
(A) Right buccal mucosa at base-line showing OLP lesions with lace-like appearance. (B) Left buccal mucosa with atrophic and reticular OLP lesions. (C, D) Improvement of lichenoid lesions with NASH treatment.

Fig. 2
(A) Histological features of the oral mucosa biopsy showing hyperorthokeratosis, basal cell damage and a band-like inflammatory infiltrate (haematoxylin–eosin, ×200) mainly composed of (B) T lymphocytes (CD3 positive, ×400) with (C) scattered B lymphocytes (CD20 positive, ×400) and (D) few plasma cells (dba 44, ×400).

A routine liver screen showed a marked alteration of the transaminase values (aspartate aminotransferase 339, alanine aminotransferase 638, gamma glutamyltransferase 247), with no signs of a past or present HBV or HCV infection (hepatitis B surface antigen negative, hepatitis B surface antibody negative, hepatitis B core antibody negative, anti-HCV antibody negative (enzyme linked immunosorbent assay and recombinant immunoblot assay III)).

The patient was referred to the Department of Gastroenterology and Hepatology (University of Turin) for a more accurate evaluation and for more laboratory tests to obtain a precise diagnosis.

The patient had a family history of diabetes, high blood pressure and hypercholesterolaemia but no history of hepatopathies. He did not consume alcohol and no exposure to hepatotoxic substances was reported. He had a history of duodenal ulcer treated with homeoprazole. Hepatic fine needle biopsy revealed macrovesicular steatosis with a slight chronic portal inflammatory infiltrate and signs of siderosis (15% of the hepatocytes were stained blue by Perls staining used to show haemosiderin accumulation) ( Fig. 3 ).

Feb 7, 2018 | Posted by in Oral and Maxillofacial Surgery | Comments Off on Non-alcoholic steatohepatitis (NASH) and oral lichen planus: a rare occurrence
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